Literature DB >> 32566130

Toward a paradigm shift from deficit-based to proactive speech and language treatment: Randomized pilot trial of the Babble Boot Camp in infants with classic galactosemia.

Beate Peter1,2, Nancy Potter3, Jennifer Davis1, Inbal Donenfeld-Peled1, Lizbeth Finestack4, Carol Stoel-Gammon5, Kari Lien1, Laurel Bruce1, Caitlin Vose6, Linda Eng1, Hanako Yokoyama1, Daniel Olds3, Mark VanDam3.   

Abstract

Background: Speech and language therapy is typically initiated reactively after a child shows delays. Infants with classic galactosemia (CG), a metabolic disease with a known high risk for both speech and language disorders, hold the keys towards evaluating whether preventive treatment is effective when the risks are known at birth. We present pilot data from a randomized parallel trial of an innovative proactive speech and language intervention program, the Babble Boot Camp (BBC).  Method: Five children with CG, otherwise healthy, participated in the study from approximately 2 to 24 months of age. One of these was randomly selected as control receiving conventional management, which typically starts at age 2-3 years. A pediatric speech-language pathologist met weekly via telepractice with the parents in the treatment cohort. Parents implemented the prespeech, speech, and language stimulation and expansion activities according to the intervention protocol. The control child was still too young for conventional treatment. Primary outcome measures were speech sound production complexity in babble and speech and expressive vocabulary size. Secondary outcome measures were vocalization rates and developmental milestones in communication, motor, and cognition. The trial is ongoing.
Results:   All four treated children had higher speech sound skills in babble, three had higher speech sound skills in meaningful speech, two had higher expressive vocabularies, three had higher global developmental scores, and two had higher vocalization rates, compared to the control child with CG. Discussion: Given the high risk for speech and language delays in children with CG, finding on-schedule abilities in two or more of the treated children but not the untreated child is unexpected under random conditions. The trends toward beneficial effects of the BBC on speech sound production, expressive language, and communication milestones warrant appropriately powered larger clinical trials with full randomization. Trial registration: ClinicalTrials.gov NCT03838016 (12 th February 2019). Copyright:
© 2020 Peter B et al.

Entities:  

Keywords:  genetic risk; infant; language impairment; prevention; speech disorder; very early intervention

Mesh:

Year:  2019        PMID: 32566130      PMCID: PMC7291108.5          DOI: 10.12688/f1000research.18062.5

Source DB:  PubMed          Journal:  F1000Res        ISSN: 2046-1402


  42 in total

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2.  Long-term prognosis in galactosaemia: results of a survey of 350 cases.

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Review 5.  Children's language learning: an interactionist perspective.

Authors:  R S Chapman
Journal:  J Child Psychol Psychiatry       Date:  2000-01       Impact factor: 8.982

6.  Randomized, controlled trial of an intervention for toddlers with autism: the Early Start Denver Model.

Authors:  Geraldine Dawson; Sally Rogers; Jeffrey Munson; Milani Smith; Jamie Winter; Jessica Greenson; Amy Donaldson; Jennifer Varley
Journal:  Pediatrics       Date:  2009-11-30       Impact factor: 7.124

7.  Correlates of language impairment in children with galactosaemia.

Authors:  N L Potter; J-A C Lazarus; J M Johnson; R D Steiner; L D Shriberg
Journal:  J Inherit Metab Dis       Date:  2008-07-12       Impact factor: 4.982

8.  Individual differences in phonological development: ages one and three years.

Authors:  M M Vihman; M Greenlee
Journal:  J Speech Hear Res       Date:  1987-12

9.  Better reporting of interventions: template for intervention description and replication (TIDieR) checklist and guide.

Authors:  Tammy C Hoffmann; Paul P Glasziou; Isabelle Boutron; Ruairidh Milne; Rafael Perera; David Moher; Douglas G Altman; Virginia Barbour; Helen Macdonald; Marie Johnston; Sarah E Lamb; Mary Dixon-Woods; Peter McCulloch; Jeremy C Wyatt; An-Wen Chan; Susan Michie
Journal:  BMJ       Date:  2014-03-07

10.  How to assess and compare inter-rater reliability, agreement and correlation of ratings: an exemplary analysis of mother-father and parent-teacher expressive vocabulary rating pairs.

Authors:  Margarita Stolarova; Corinna Wolf; Tanja Rinker; Aenne Brielmann
Journal:  Front Psychol       Date:  2014-06-04
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  6 in total

1.  Translating principles of precision medicine into speech-language pathology: Clinical trial of a proactive speech and language intervention for infants with classic galactosemia.

Authors:  Beate Peter; Jennifer Davis; Lizbeth Finestack; Carol Stoel-Gammon; Mark VanDam; Laurel Bruce; Yookyung Kim; Linda Eng; Sarah Cotter; Emily Landis; Sam Beames; Nancy Scherer; Ina Knerr; Delaney Williams; Claire Schrock; Nancy Potter
Journal:  HGG Adv       Date:  2022-05-20

2.  Infants' neural speech discrimination predicts individual differences in grammar ability at 6 years of age and their risk of developing speech-language disorders.

Authors:  T Christina Zhao; Olivia Boorom; Patricia K Kuhl; Reyna Gordon
Journal:  Dev Cogn Neurosci       Date:  2021-03-31       Impact factor: 5.811

Review 3.  Current and Future Treatments for Classic Galactosemia.

Authors:  Britt Delnoy; Ana I Coelho; Maria Estela Rubio-Gozalbo
Journal:  J Pers Med       Date:  2021-01-28

Review 4.  Tools for standardized data collection: Speech, Language, and Hearing measurement protocols in the PhenX Toolkit.

Authors:  Cynthia C Morton; Mary L Marazita; Beate Peter; Mabel L Rice; Shelly Jo Kraft; Julie Barkmeier-Kraemer; Carey Balaban; Michael Phillips; Jennifer Schoden; Deborah Maiese; Tabitha Hendershot; Carol M Hamilton
Journal:  Ann Hum Genet       Date:  2021-09-28       Impact factor: 2.180

5.  Toward Preventing Speech and Language Disorders of Known Genetic Origin: First Post-Intervention Results of Babble Boot Camp in Children With Classic Galactosemia.

Authors:  Beate Peter; Jennifer Davis; Sarah Cotter; Alicia Belter; Emma Williams; Melissa Stumpf; Laurel Bruce; Linda Eng; Yookyung Kim; Lizbeth Finestack; Carol Stoel-Gammon; Delaney Williams; Nancy Scherer; Mark VanDam; Nancy Potter
Journal:  Am J Speech Lang Pathol       Date:  2021-10-19       Impact factor: 4.018

Review 6.  Galactosemia: Biochemistry, Molecular Genetics, Newborn Screening, and Treatment.

Authors:  Mariangela Succoio; Rosa Sacchettini; Alessandro Rossi; Giancarlo Parenti; Margherita Ruoppolo
Journal:  Biomolecules       Date:  2022-07-11
  6 in total

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